Unresectable giant cell tumor of cervical spine

Suresh S Pillai; Premdeep Dennison; TV Rishin; Jim Thomas Malayil

doi:10.4103/joasis.joasis_20_22

CASE SERIES

Year : 2022 | Volume : 19 | Issue : 1 | Page : 48-50

Unresectable giant cell tumor of cervical spine

Suresh S Pillai¹, Premdeep Dennison¹, TV Rishin¹, Jim Thomas Malayil²
¹ Department of Spine Surgery, Baby Memorial Hospital, Kozhikode, Kerala, India
² Department of Orthopaedics, Baby Memorial Hospital, Kozhikode, Kerala, India

Date of Web Publication

26-Jun-2022

Correspondence Address:
Premdeep Dennison
Department of Spine Surgery, Baby Memorial Hospital, Kozhikode, Kerala
India

Source of Support: None, Conflict of Interest: None

DOI: 10.4103/joasis.joasis_20_22

Abstract

Giant cell tumors (GCTs) of the spine occurs very rarely above the sacrum. They are even rarer in cervical spine. The complete resection of the tumor is often unachievable in cervical spine due to the close proximity of important neurovascular structures. We describe a case of unresectable (GCT) of cervical spine treated with near-total resection and adjuvant denosumab therapy. A 28-year-old female presented with quadriparesis and imaging showed GCT of the C6 vertebral body, compressing the spinal cord and also abutting the vertebral artery on the left side. Vertebral arteries were dominant on both sides and the feeders to the tumor could not be embolized. This posed a major challenge as removal of the tumor was not feasible. Near-total removal was done and the patient was started on denosumab, which decreases the osteoclast production and thus reduces size and prevents further spread of tumor. GCTs are resilient and can recur locally, even after the complete removal of tumor. Denosumab is a novel monoclonal antibody which can decrease the size of the tumor and also decrease the chance of recurrence after surgery, by preventing osteoclastogenesis. In cases of GCTs which cannot be completely removed, denosumab therapy as an adjunct to surgical removal is a safe and reliable option.

Keywords: Cervical spine, denosumab, giant cell tumor

How to cite this article:
Pillai SS, Dennison P, Rishin T V, Malayil JT. Unresectable giant cell tumor of cervical spine. J Orthop Assoc South Indian States 2022;19:48-50

How to cite this URL:
Pillai SS, Dennison P, Rishin T V, Malayil JT. Unresectable giant cell tumor of cervical spine. J Orthop Assoc South Indian States [serial online] 2022 [cited 2023 Apr 1];19:48-50. Available from: https://www.joasis.org/text.asp?2022/19/1/48/348319

Introduction

Giant cell tumors (GCTs) are benign, locally aggressive primary tumors of bone. They usually affect the epiphysis of long bones. They are extremely rare in cervical spine. They are resilient and have a tendency to recur at the same site even after excision. Hence, they are treated with complete resection followed by extended curettage using chemical, thermal, or mechanical agents. Complete resection is often not possible due to the proximity of important neurovascular structures. Radiotherapy or monoclonal antibody therapy can be used postoperatively to prevent recurrence. Radiotherapy carries malignant potential and is not advisable. Denosumab is a novel monoclonal antibody which acts by binding to receptor activator of nuclear factor kappa-β ligand (RANKL) and directly preventing osteoclastogenesis.

We describe a case of unresectable GCT of cervical spine, the decision-making process, and the course of management. The purpose of the case report is to outline a feasible course of treatment in cases of unresectable GCT of the vertebra.

Case Report

A 28-year-old female came to outpatient department with a history of neck pain and weakness of the left upper limb and both lower limbs. She was wheelchair-bound. X-ray showed a lytic lesion on the C5 vertebra. Magnetic resonance imaging of cervical spine showed near complete collapse of the C5 vertebra with erosion of the C4 inferior endplate and involvement of transverse process associated with soft tissue compromising the vertebral foramen. She was further evaluated with fludeoxyglucose positron emission tomography scan, which showed only local erosion of C4 and a benign fibroadenoma of the breast.

With a possible diagnosis of GCT of the C5 vertebral body, removal and biopsy, after preoperative embolization of the lesion, were planned. Computed tomography of neck and intracranial vessel angiogram showed both vertebral arteries are dominant and could not be embolized. The hyperdense component was also noted as abutting the left vertebral artery at the C5 level. An anterior approach was adopted for the removal of the lesion. Intraoperative frozen section was done and tissue diagnosis of GCT was confirmed before proceeding with near-total removal. Removal was done until the vertebral artery was visible and no further removal could be attempted without compromising the integrity of the artery. Further removal was not attempted as the tumor started bleeding heavily. A right iliac crest bone graft was applied to the defect. Fixation was done with cervical spine locking plate and screws, from C4 to C6.

Philadelphia collar was applied postoperatively for additional immobilization. Biopsy of the lesion confirmed the diagnosis of GCT. Postoperatively denosumab therapy was initiated on a monthly frequency. She is able to walk normally with complete neurological recovery postoperatively.

Discussion

GCTs of spine occurs very rarely above the sacrum. They are even rarer in cervical spine. GCTs are locally aggressive, metabolically benign neoplasms that usually involve the metaphysis and epiphysis of long bones. The aggressiveness of GCT can differ between individuals and can cause local destruction and metastasis. Studies have reported GCT metastasis to the lymph nodes, liver, soft tissue, brain, mediastinum, scalp, kidney, and penis.^[1]

Histologically GCT is characterized by many multinucleated osteoclast-type cells in a background of oval or plump, spindle-shaped uninucleated cells. According to Bhaker et al., the frozen sections and paraffin-embedded sections yielded the same diagnoses in 91.7% of cases. Tissue imprinting is also reported as an useful tool in obtaining intraoperative diagnosis in musculoskeletal tumors.^[1],[2]

Griessenauer et al. after a large meta-analysis concluded embolization of spinal tumors as a safe and efficacious treatment adjunct before surgery. It decreases intraoperative blood loss and facilitates tumor resection and is usually done <72 h before surgery.^[3]

Surgical treatment options include curettage using high-speed burr or resection. Curettage has a high recurrence rate, but maintains the relationship intact with adjacent structures. Tumor recurrence can be reduced by resection with a wide margin but functional outcomes are bad.^[4] Radiotherapy carries a risk of malignant transformation to sarcoma and can cause myelopathy.^[1] With surgery, the recurrence rate of GCT is 25%–45%.^[4]

Denosumab is a monoclonal antibody that binds to the RANKL. GCT stromal cells express RANKL and osteoclast-like giant cells express RANK. Denosumab binds to RANKL and substantially decreases or completely eliminates osteoclast-like giant cells. Thus, denosumab suppresses osteolysis and proliferative tumor stroma is replaced with nonproliferative, differentiated, densely woven new bone.^[5] Chawla et al. established the safety and efficacy of denosumab in the treatment of GCT. Denosumab is administered as a subcutaneous injection of 120 mg every 4 weeks.^[6] Common adverse effects of the therapy included arthralgia, headache, nausea, fatigue, back pain, and extremity pain. Major adverse effects were osteonecrosis of the jaw (2%), hypocalcemia (5% – none serious), new primary malignancy (1%), and infections (1%). Studies suggest that denosumab therapy 6 months before surgery, reduces the size of the tumour – which makes it easier to be resected and thus decreases complications and morbidity. The recurrence rate of surgery coupled with denosumab therapy was reported to be 2%.^[4],[7] The major drawback as far as patients are concerned is the prolonged therapy required. The present studies suggest that lifelong therapy is needed.^[5] The safety of denosumab in the immature skeleton is also a concern.

Conclusion

GCT of cervical spine is a very rare tumor but carries significant challenges in management due to the proximity of vital neurovascular structures. In cases of GCTs, which cannot be completely removed, denosumab therapy as an adjunct to surgical removal is a safe and reliable option.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

References

1.	Hunter CL, Pacione D, Hornyak M, Murali R. Giant-cell tumors of the cervical spine: Case report. Neurosurgery 2006;59:E1142-3.
2.	Bhaker P, Mohan H, Handa U, Kumar S. Role of Intraoperative Pathology Consultation in Skeletal Tumors and Tumor-Like Lesions. Sarcoma. 2014 May 19;2014:e902104
3.	Griessenauer CJ, Salem M, Hendrix P, Foreman PM, Ogilvy CS, Thomas AJ. Preoperative embolization of spinal tumors: A systematic review and meta-analysis. World Neurosurg 2016;87:362-71.
4.	Jamshidi K, Gharehdaghi M, Hajialiloo SS, Mirkazemi M, Ghaffarzadehgan K, Izanloo A. Denosumab in patients with giant cell tumor and its recurrence: A systematic review. Arch Bone Jt Surg 2018;6:260-8.
5.	Branstetter DG, Nelson SD, Manivel JC, Blay JY, Chawla S, Thomas DM, et al. Denosumab induces tumor reduction and bone formation in patients with giant-cell tumor of bone. Clin Cancer Res 2012;18:4415-24.
6.	Yayan J. Denosumab for effective tumor size reduction in patients with giant cell tumors of the bone: A systematic review and meta-analysis. Cancer Control 2020;27:1073–2748.
7.	Chawla S, Henshaw R, Seeger L, Choy E, Blay JY, Ferrari S, et al. Safety and efficacy of denosumab for adults and skeletally mature adolescents with giant cell tumour of bone: Interim analysis of an open-label, parallel-group, phase 2 study. Lancet Oncol 2013;14:901-8.